Razavi Int J Med. 2015 May; 3(2): e21183.
DOI: 10.5812/rijm.21183
Case Report
Published online 2015 May 2.
A Rare Osteoarticular Brucellosis in Gonabad City, Iran: A Case Report
1
Hamed Ramezani Awal Riabi ; Reza Ahmadi
1,*
1Department of Basic Sciences, School of Public Health, Gonabad University of Medical Sciences, Gonabad, IR Iran
*Corresponding author: Reza Ahmadi, Department of Basic Sciences, School of Public Health, Gonabad University of Medical Sciences, Gonabad, IR Iran. Tel: +98-5337223514,
E-mail: [email protected]
Received: September 3, 2014; Revised: November 25, 2014; Accepted: December 17, 2014
Introduction: Species of bacteria brucellosis, which can infect humans, are Brucella abortus, B melitensis, B. suis and rarely B. canis.
Case Presentation: A 71-year-old woman with severe pain in the limbs, especially the left pelvic pain was admitted to 22-Bahman hospital
in Gonabad, Iran. The patient’s Erythrocyte Sedimentation Rate (ESR) was 100 and C-reactive protein (CRP) had positive finding. Ureacreatinine, liver enzymes, U/A and U/C had normal results. A signal change in the soft tissue adjacent to the posterior surface of the vertebral
body L4-L5 was observed. According to Wright and 2 Mercaptoethanol (ME) negative results, patient was re-introduced to neurosurgeon.
Due to severe bone pain, radiotherapy was stopped and she was referred for treatment with bone tuberculosis diagnosis to a health center
in Gonabad city, Iran. Daily streptomycin was started for patient and after 10 days, she referred to health center with a good general health
status and without pelvic pain.
Conclusions: Prominent musculoskeletal complaints (especially back pain) accompanied by constitutional symptoms such as fever,
malaise and weight loss may be consistent with brucellosis.
Keywords: Diagnosis; brucellosis; case report; Iran
1. Introduction
We reported here a woman with brucellar spin arthritis who had only joint complaints without any other systemic symptoms. Spin arthritis can occur in Brucellosis,
but there were a few studies and case reports about this
subject. Brucella is a Gram-negative, Coco bacillus, small
and non-spore Bacterium. Species that can infect humans
are Brucella abortus, B. melitensis, B. suis and rarely B. canis.
Brucellosis is one of the most serious health and economic problems in most parts of the world (1, 2).
Brucella can infect every organ system of humans, including endocardium, meninges, epididymis, testes,
liver, bones, spleen, etc. (3). Humans as accidental hosts
can be infected with Brucella. Another name for brucellosis in cattle is contagious abortion or the Bang's disease.
In Iran, the first Gram-negative bacteria were isolated in
1932 from a patient's blood. Among strains of causative
agent of brucellosis, Brucella melitensis biovar 1 strains are
the most common strains of human brucellosis. Melitensis has been isolated from goats and sheep. Brucella can
infect many organs and tissues in the human body. Incidence of brucellosis is low in developed countries, but it
has been seen sporadic in occupational groups such as
farmers, veterinarians, laboratories and abattoirs workers. It is native to Iran.
Genital brucellosis, a localized complex brucellosis contains 2 - 20% of human Brucella (4). Brucella species were
first described as a cause of granulomatous orchitis in
humans by Bardy in 1928 (5). Diagnosis of human brucel-
losis relies on serological tests such as Standard Agglutination Test (SAT), Enzyme-Linked immunosorbent Assay
(ELISA) and Combs Test (2).
2. Case Presentation
A 71-year-old woman with severe pain in the limbs, especially the left pelvic pain was admitted to 22-Bahman
hospital in Gonabad, Iran. Discomfort started 15 days
prior to admission with fever, musculoskeletal pain and
anorexia. The patient’s medical history did not have any
specific drug use or disease. In general examination, the
patient was febrile and complained of severe pain in the
pelvic area, which did not respond to usual narcotic medicine. The laboratory results of patient were white blood
cell count of 9700 per cubic milliliter, 90% neutrophils,
6% lymphocytes, hemoglobin 8.1 and hematocrit 6.27,
respectively. ESR was 100 and CRP (C-reactive protein) 2
plus, also Wright, Coombs Wright and 2 ME had negative
results. Urea creatinine and liver enzymes had normal
findings. U/A and U/C were in the normal range. Chest Xray (CXR) showed diffuse nodular opacities in both lungs.
MRI showed a reduction of signals in the vertebral discs
because of degeneration changes. A signal change was observed in the soft tissue adjacent to the posterior surface
of the vertebral body at L4-L5 (Figure 1). Due to negative
results of Wright and 2 ME, the patient was referred back
to neurosurgeon. The second MRI revealed degenerative
Copyright © 2015, Razavi Hospital. This is an open-access article distributed under the terms of the Creative Commons Attribution-NonCommercial 4.0 International License (http://creativecommons.org/licenses/by-nc/4.0/) which permits copy and redistribute the material just in noncommercial usages, provided the original
work is properly cited.
Ramezani Awal Riabi H et al.
Figure 1. Signal Increase in the Posterior L4 and L5 and S1 Spinal Fluid Can Represent Infectious Process of Brucellosis
Figure 2. Change of Signal in Sagittal Sections of Sequence T2 IR-TSE Caused by Infectious Spondylitis Shows Diffuse Increased Signal Intensity in the
Lumbar 4 and Paravertebral Soft Tissues of the Spine 3-5
changes in spine, L4 and L5 (Figure 2). A bone biopsy was
performed for patient, and by diagnosis of bone tumors,
she completed 10 radiotherapy sessions. However, due
to remaining severe bone pain, especially pelvic joint,
the patient referred to an orthopedic specialist in Mashhad by diagnosis of bone tuberculosis, radiotherapy was
stopped and the patient was referred to the health center
in Gonabad, Iran to continue treatment. In a review of
medical records of the patient by a specialist in infectious
diseases (the corresponding author of this article), it was
observed that brucellosis tests of the patient in 12/3/2012
showed positive results for Wright as 1/80, 2 ME 1/40 and
Coombs Wright 1/80. Therefore, treatment began with Rifampin 600 mg, Doxycycline and Streptomycin daily. Af2
ter 10 days, the patient referred to the health center with
a good general health status and without pelvic pain.
3. Conclusions
This was the first report of brucellosis bone in Gonabad
city. Brucellosis is endemic in Mediterranean countries
and West Asia. Thus, the disease in most provinces of Iran
is a native disease (5). In Gonabad, located in Khorasan
Razavi province, the incidence of brucellosis was 113 cases
per 100000 populations in 2012, while it was 19 cases per
100000 persons in the country. This higher incidence
might be due to abundant livestock in the villages and
non-vaccinated animals entering city that spread brucelRazavi Int J Med. 2015;3(2):e21183
Ramezani Awal Riabi H et al.
losis in livestock, particularly sheep, leading to abortion
in them (6).
Skeletal brucellosis is the most widespread brucellosis seen in approximately 85% of patients. Three forms
of musculoskeletal brucellosis are sacroiliitis, spondylitis and peripheral arthritis. The skeleton is a dynamic
physical system, being renovated constantly. These steps
include coordinating the efforts of osteoblasts and osteoclasts. The performances of the two cells together represent stability and strength of bone (7).
In 10 - 25% of patients, the symptom of Brucella infection
is arthritis. The most commonly sites of arthritis are knee
(25%), hip (18%), spine (8%) and sacroiliac joint (26%). Osteoarticular brucellosis was documented by Marston in
1861 at the time when this disease had not even existed
as a separate entity (8). Peripheral arthritis had been described as the commonest form of osteoarticular complications in acute brucellosis (9).
In the study of 104 cases of brucellosis in Saudi Arabia
and 1288 cases in the United States, the most common
symptoms reported were arthritis, lymphadenopathy,
weakness, fever, backache, chills, malaise, sweating,
headache and splenomegaly (10).
In another report analysis of joint fluid, B. Melitensis was
isolated from a 49-year-old farmer with knee pain for 20
years and had not been under any treatment. Antimicrobial therapy with Doxycycline, Rifampin and Ciprofloxacin was performed for six months and he was treated (3).
In other study, researchers reported and treated a multifocal osteomyelitis diagnosed as chronic brucellosis
who had for 14 years (3). In a similar study, a 10-year-old
boy from a village in southeastern Turkey was admitted
with painful limited motion of right lower limb for two
weeks. There was no history of trauma and no family history of inflammatory joint disease. The laboratory results
showed erythrocyte sedimentation rate (ESR) as 72 mm/h
(millimeters per hour), (CRP): 18 mg/L leukocyte count of
8.500/mm3, negative antinuclear antibody, antistreptolysin (ASO) and rheumatoid factor within the normal limits, blood urea levels, creatinine levels and liver function
tests as normal. Coombs Wright was performed twice
with an interval of three days. The results were positive
with titers of 1/320 and 1/640 (11).
Although nonspecific clinical signs and symptoms
(such as fever or arthralgias) often make the clinical diagnosis difficult, the frequency and characteristic patterns
of localized disease should heighten clinicians' index of
Razavi Int J Med. 2015;3(2):e21183
suspicion and lower the threshold for a serologic investigation. Prominent musculoskeletal complaints (especially back pain) accompanied by constitutional symptoms
such as fever, malaise and weight loss may be consistent
with brucellosis.
Acknowledgements
The authors wish to thank all coworkers of 22-Bahman
hospital of Gonabad.
Authors’ Contributions
Hamed Ramezani Awal Riabi: study concept and design;
Reza Ahmadi, analysis and interpretation.
Funding/Support
This study was financially supported by the Research
Council of the Medical Faculty of Gonabad University of
Medical Sciences.
References
1.
2.
3.
4.
5.
6.
7.
8.
9.
10.
11.
Salari MH, Khalili MB, Hassanpour GR. Selected epidemiological
features of human brucellosis in Yazd, Islamic Republic of Iran:
1993-1998. East Mediterr Health J. 2003;9(5-6):1054–60.
Zeinali M. National Guideline for Brucellosis Control, CDC.; 2010.
Yorgancigil H, Yayli G, Oyar O. Neglected case of osteoarticular
Brucella infection of the knee. Croat Med J. 2003;44(6):761–3.
Motaharinia Y, Rezaee MA, Zandi H, Hazhir MS, Shapouri R, Hakhamaneshi MS, et al. Evaluation of the Antibacterial Activity of Zataria Multifl ora Boiss., Rhus Coriaria L. (sumac), Mentha Piperita
L., and Ocimum Basilicum L. Extracts on Brucella Strains Isolated
From Brucellosis Patients. Turk J Med Sci. 2012;42(5):816–22.
Najafi N, Ghassemian R, Davoody AR, Tayebi A. An unusual complication of a common endemic disease: clinical and laboratory
aspects of patients with brucella epididymoorchitis in the north
of Iran. BMC Res Notes. 2011;4:286.
Centers for Disease Control and Prevention.. Annual Reportet of
Centers for Disease Control and Prevention (CDC). 2013. Available
from: http://www.amlin.com.
Scian R, Barrionuevo P, Fossati CA, Giambartolomei GH, Delpino
MV. Brucella abortus invasion of osteoblasts inhibits bone formation. Infect Immun. 2012;80(7):2333–45.
Solak Y, Biyik Z, Demircioglu S, Polat I, Genc N, Turkmen K, et al.
Brucella peritonitis in peritoneal dialysis: a case report and review of the literature. Perit Dial Int. 2012;32(2):126–30.
al-Eissa YA, Kambal AM, Alrabeeah AA, Abdullah AM, al-Jurayyan NA, al-Jishi NM. Osteoarticular brucellosis in children. Ann
Rheum Dis. 1990;49(11):896–900.
Salari MH. Seroepidemiological Survey of Brucellosis Among Animal Farmers of Yazd Province . Iranian J. Publ. Health. 2002;31(12):29–32.
Altindag O, Sirmatel F, Sirmatel O. Brucella Arthritis of Hip: A
Case Report, Romatizma. Rheumatism. 2007;22:40–2.
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